| Title | Inherited IFNAR1 deficiency in otherwise healthy patients with adverse reaction to measles and yellow fever live vaccines. |
| Publication Type | Journal Article |
| Year of Publication | 2019 |
| Authors | Hernandez N, Bucciol G, Moens L, Le Pen J, Shahrooei M, Goudouris E, Shirkani A, Changi-Ashtiani M, Rokni-Zadeh H, Sayar EHazar, Reisli I, Lefevre-Utile A, Zijlmans D, Jurado A, Pholien R, Drutman S, Belkaya S, Cobat A, Boudewijns R, Jochmans D, Neyts J, Seeleuthner Y, Lorenzo-Diaz L, Enemchukwu C, Tietjen I, Hoffmann H-H, Momenilandi M, Pöyhönen L, Siqueira MM, de Lima SMBarbosa, Matos DC de Souza, Homma A, Maia Mde Lourdes, Barros TAzamor da, de Oliveira PMouta Nune, Mesquita ECiclini, Gijsbers R, Zhang S-Y, Seligman SJ, Abel L, Hertzog P, Marr N, Martins Rde Menezes, Meyts I, Zhang Q, MacDonald MR, Rice CM, Casanova J-L, Jouanguy E, Bossuyt X |
| Journal | J Exp Med |
| Volume | 216 |
| Issue | 9 |
| Pagination | 2057-2070 |
| Date Published | 2019 Sep 02 |
| ISSN | 1540-9538 |
| Abstract | Vaccination against measles, mumps, and rubella (MMR) and yellow fever (YF) with live attenuated viruses can rarely cause life-threatening disease. Severe illness by MMR vaccines can be caused by inborn errors of type I and/or III interferon (IFN) immunity (mutations in , , or ). Adverse reactions to the YF vaccine have remained unexplained. We report two otherwise healthy patients, a 9-yr-old boy in Iran with severe measles vaccine disease at 1 yr and a 14-yr-old girl in Brazil with viscerotropic disease caused by the YF vaccine at 12 yr. The Iranian patient is homozygous and the Brazilian patient compound heterozygous for loss-of-function variations. Patient-derived fibroblasts are susceptible to viruses, including the YF and measles virus vaccine strains, in the absence or presence of exogenous type I IFN. The patients' fibroblast phenotypes are rescued with WT Autosomal recessive, complete IFNAR1 deficiency can result in life-threatening complications of vaccination with live attenuated measles and YF viruses in previously healthy individuals. |
| DOI | 10.1084/jem.20182295 |
| Alternate Journal | J. Exp. Med. |
| PubMed ID | 31270247 |
| PubMed Central ID | PMC6719432 |
| Grant List | R01 AI124690 / AI / NIAID NIH HHS / United States T32 GM066699 / GM / NIGMS NIH HHS / United States |
Submitted by api_import on December 2, 2019 - 10:06am